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JOURNAL ARTICLE
Meziane Brizini, Bruno Michon, Marc-Antoine Bédard
Multiple myeloma is a rare disease in pediatrics, where about 30 cases are described under 15 years old. It is even rarer when atypical multiple myeloma occurs in the context of autoimmunity. This case describes a 9-year-old female with autoimmune lymphoproliferative-like disease and combined immune deficiency that developed acute kidney failure with monoclonal peak associated with RAC2 and TNFRSF9 variants. An adapted protocol from the backbone adult multiple myeloma standard of care with the addition of an allogeneic hematopoietic stem cell transplant was used...
April 23, 2024: Journal of Pediatric Hematology/oncology
#2
JOURNAL ARTICLE
Vasil Toskov, Annamaria Cseh, Alexander Claviez, Beatrice Drexler, Natalia Rotari, Stephan Schwarz-Furlan, Matthias Braun, Peter Bader, Peter Lang, Rita Beier, Bernhard Erdlenbruch, Monika Führer, Miriam Erlacher, Charlotte M Niemeyer, Brigitte Strahm, Ayami Yoshimi
We report 5 children with bone marrow failure (BMF) after primary varicella zoster virus (VZV) infection or VZV vaccination, highlighting the highly variable course. Two patients were treated with intravenous immunoglobulins; one had a slow hematologic recovery, and the other was rescued by allogeneic hematopoietic stem cell transplantation (HSCT). Of the 2 patients treated with immunosuppressive therapy with antithymocyte globulin and cyclosporine, one had a complete response, and the other was transplanted for nonresponse...
April 23, 2024: Journal of Pediatric Hematology/oncology
#3
JOURNAL ARTICLE
Yao Li, Guan-Hua Hu, Lan-Ping Xu, Xiao-Hui Zhang, Kai-Yan Liu, Pan Suo, Yu Wang, Yi-Fei Cheng, Xiao-Jun Huang
BACKGROUND: Philadelphia chromosome (Ph)-positive B-cell acute lymphoblastic leukemia (ALL) has a high complete remission (CR) rate, but relapse and prolonged measurable residual disease remain serious problems. We sought to describe the CR rate measurable residual disease negative rate and address the results and safety of pediatric patients who underwent after receiving chimeric antigen receptor (CAR) specific for CD19 (CAR-19) followed by hematopoietic stem cell transplantation (HSCT) for the treatment of Ph-positive ALL...
April 23, 2024: Journal of Pediatric Hematology/oncology
#4
JOURNAL ARTICLE
Kathryn A Six, Inmaculada B Aban, Gabriel M Daniels, Julie Wolfson, Elizabeth A Beierle, Matthew A Kutny, Jeffrey Lebensburger, Ana C Xavier
Lymphadenopathy is a common reason for referral to a subspecialist, which may result in significant anxiety for parents. Understanding which patients require a subspecialty referral for lymphadenopathy is key to streamlining health care utilization for this common clinical entity. This is an IRB-approved retrospective study examining pediatric patients consecutively referred to pediatric hematology oncology, otolaryngology, or surgery for lymphadenopathy from 2012 to 2021 at a free-standing tertiary-care children's hospital...
April 23, 2024: Journal of Pediatric Hematology/oncology
#5
Delcroix Luca, Tamburello Sara, Innocenti Marco, Campanacci Domenico Andrea
BACKGROUND: Post-oncologic surgical reconstruction of lower limbs in pediatrics remains a challenging topic. Microsurgical techniques allow reconstructions of large bony defects. The use of vascularized fibular flap with allograft has proven to be an ideal biologic construct. We aim to assess the success rate of this operation, including flap survival, bony union, weight-bearing ambulation, and complications in a long-term follow-up in our case series compared to the literature. PATIENTS AND METHODS: Our case-series includes 18 femoral resections (9 osteosarcomas, 8 Ewing sarcoma, and 1 desmoid tumor) and 15 tibial resections (10 osteosarcoma, 4 Ewing sarcoma, and 1 Malignant Fibrous Histiocytoma)...
May 2024: Microsurgery
#6
Meenakshi Gopalakrishnan, Arunalini Ramanathan, Dhaarani Jayaraman, Sri Gayathri Shanmugam, Julius Xavier Scott
Hemophagocytic lymphohistiocytosis (HLH) is a rare, life-threatening hematological disorder of immune dysregulation associated with significant challenges in diagnosis and management. Described as primary HLH secondary to genetic defects or more commonly secondary to infections, it can also occur secondary to malignancy, i.e., malignancy-associated hemophagocytic lymphohistiocytosis (M-HLH). A five-year-old male child presented with left cervical adenopathy and a high-spiking fever for two weeks. He had pallor, anasarca, multiple enlarged and matted cervical lymph nodes, respiratory distress, and hepatomegaly...
March 2024: Curēus
#7
REVIEW
Annelot J M Meijer, Franciscus A Diepstraten, Marc Ansari, Eric Bouffet, Archie Bleyer, Brice Fresneau, James I Geller, Alwin D R Huitema, Per Kogner, Rudolf Maibach, Allison F O'Neill, Vassilios Papadakis, Kaukab M Rajput, Gareth J Veal, Michael Sullivan, Marry M van den Heuvel-Eibrink, Penelope R Brock
PURPOSE: Hearing loss occurs in 50%-70% of children treated with cisplatin. Scientific efforts have led to the recent approval of a pediatric formula of intravenous sodium thiosulfate (STS) for otoprotection by the US Food and Drug Administration, the European Medicines Agency, and the Medicines and Health Regulatory Authority in the United Kingdom. To inform stakeholders regarding the clinical utility of STS, the current review summarizes available literature on the efficacy, pharmacokinetics (PK), and safety of systemic STS to minimize cisplatin-induced hearing loss (CIHL)...
April 22, 2024: Journal of Clinical Oncology
#8
REVIEW
James A Cahill, Leslie A Smith, Soumya Gottipati, Tina Salehi Torabi, Kiley Graim
Dogs are humanity's oldest friend, the first species we domesticated 20,000-40,000 years ago. In this unequaled collaboration, dogs have inadvertently but serendipitously been molded into a potent human cancer model. Unlike many common model species, dogs are raised in the same environment as humans and present with spontaneous tumors with human-like comorbidities, immunocompetency, and heterogeneity. In breast, bladder, blood, and several pediatric cancers, in-depth profiling of dog and human tumors has established the benefits of the dog model...
April 22, 2024: Annual review of biomedical data science
#9
JOURNAL ARTICLE
Anna Laura Knoblauch, B-I Blaß, C Steiert, N Neidert, A Puzik, E Neumann-Haefelin, A Ganner, F Kotsis, T Schäfer, H P H Neumann, S Elsheikh, J Beck, J-H Klingler
PURPOSE: Von Hippel-Lindau (VHL) disease is an autosomal-dominantly inherited tumor predisposition syndrome. One of the most common tumors are central nervous system (CNS) hemangioblastomas. Recommendations on the initiation and continuation of the screening and surveillance program for CNS tumors in pediatric VHL patients are based on small case series and thus low evidence level. To derive more robust screening recommendations, we report on the largest monocentric pediatric cohort of VHL patients...
April 22, 2024: Journal of Neuro-oncology
#10
JOURNAL ARTICLE
Alex Q Lee, Hiroaki Konishi, Masami Ijiri, Yueju Li, Arun Panigrahi, Jeremy Chien, Noriko Satake
Juvenile myelomonocytic leukemia (JMML) is an aggressive pediatric leukemia with few effective treatments and poor outcomes even after stem cell transplantation, the only current curative treatment. We developed a JMML patient-derived xenograft (PDX) mouse model and demonstrated the in vivo therapeutic efficacy and confirmed the target of trametinib, a RAS-RAF-MEK-ERK pathway inhibitor, in this model. A PDX model was created through transplantation of patient JMML cells into mice, up to the second generation, and successful engraftment was confirmed using flow cytometry...
April 22, 2024: Pediatric Hematology and Oncology
#11
Matthew T Perry, Andrew J Witten, Majeed Marwan, Alexander Vortmeyer, Jignesh Tailor
Rhabdomyosarcoma (RMS) is a pediatric malignancy with a variable prognosis depending on tumor stage and genotype. There has been a significant improvement in survival rates over the past decades. However, aggressive variants can metastasize to locations that are difficult to treat. We report a case of prostatic alveolar rhabdomyosarcoma with metastases to the bone marrow and thoracic spine in a child. The patient was treated with a multimodal approach that included surgical resection of the epidural mass; the administration of vincristine, dactinomycin, and cyclophosphamide; and radiotherapy...
March 2024: Curēus
#12
JOURNAL ARTICLE
Sujin Gang, Hyunhee Kwon, In Hye Song, Jung-Man Namgoong
BACKGROUND: Biliary intraepithelial neoplasia (BilIN), a noninvasive precursor of cholangiocarcinoma, can manifest malignant transformation. Since cholangiocarcinoma (CCA) may progress due to chronic inflammation in the bile ducts and gallbladder, choledochal cysts are considered a precursor to CCA. However, BilIN has rarely been reported in children, to date. METHODS: We reviewed medical records of patients (< 18 years of age, n = 329) who underwent choledochal cyst excision at Asan Medical Center from 2008 to 2022...
April 20, 2024: World Journal of Surgical Oncology
#13
JOURNAL ARTICLE
Larisa Gorenstein, Shai Shrot, Michal Ben-Ami, Eve Stern, Michal Yalon, Chen Hoffmann, Shani Caspi, Michal Lurye, Amos Toren, Gadi Abebe-Campino, Dalit Modan-Moses
BACKGROUND AND PURPOSE: Multiple studies demonstrated hypothalamic-pituitary dysfunction in survivors of pediatric brain tumors. However, few studies investigated the trajectories of pituitary height in these patients and their associations with pituitary function. We aimed to evaluate longitudinal changes of pituitary height in children and adolescents with brain tumors, and their association with endocrine deficiencies. MATERIALS AND METHODS: We conducted a retrospective analysis of 193 pediatric patients (54...
April 17, 2024: Radiotherapy and Oncology
#14
JOURNAL ARTICLE
Kevin J Bielamowicz, Mary Beth Littrell, Gregory W Albert, Lora S Parker, Sateesh Jayappa, Kenneth Aldape, Murat Gokden
PURPOSE: Central nervous system (CNS) embryonal tumors are a diverse group of malignant tumors typically affecting pediatric patients that recently have been better defined, and this paper describes evolution of a unique type of embryonal tumor at relapse. METHODS: Two pediatric patients with CNS embryonal tumors with EWSR1-PLAGL1 rearrangements treated at Arkansas Children's Hospital with histopathologic and molecular data are described. RESULTS: These two patients at diagnosis were classified as CNS embryonal tumors with EWSR1-PLAGL1 rearrangements based on histologic appearance and molecular data...
April 19, 2024: Journal of Neuro-oncology
#15
REVIEW
Zih-Yi Sung, Yong-Qi Liao, Jung-Hsiu Hou, Hong-Hsien Lai, Sung-Ming Weng, Hai-Wei Jao, Buo-Jia Lu, Chi-Huang Chen
Recently, there has been increasing emphasis on the gonadotoxic effects of cancer therapy in prepubertal boys. As advances in oncology treatments continue to enhance survival rates for prepubertal boys, the need for preserving their functional testicular tissue for future reproduction becomes increasingly vital. Therefore, we explore cutting-edge strategies in fertility preservation, focusing on the cryopreservation and transplantation of immature testicular tissue as a promising avenue. The evolution of cryopreservation techniques, from controlled slow freezing to more recent advancements in vitrification, with an assessment of their strengths and limitations was exhibited...
April 18, 2024: Reproductive Biology and Endocrinology: RB&E
#16
JOURNAL ARTICLE
Rodrigo L P Romao, Jennifer H Aldrink, Lindsay A Renfro, Elizabeth A Mullen, Andrew J Murphy, Jack Brzezinski, Marcus M Malek, Daniel J Benedetti, Nicholas G Cost, Ethan Smith, Jeffrey S Dome, Andrew M Davidoff, Amy Treece, Lauren N Parsons, Conrad V Fernandez, Brett Tornwall, Robert C Shamberger, Arnold Paulino, John A Kalapurakal, James I Geller, Peter F Ehrlich
INTRODUCTION: The purpose of this study is to examine the outcomes in children with anaplastic bilateral Wilms tumor (BWT) from study AREN0534 in order to define potential prognostic factors and areas to target in future clinical trials. METHODS: Demographic and clinical data from AREN0534 study patients with anaplasia (focal anaplasia [FA], or diffuse anaplasia [DA]) were compared. Event-free survival (EFS) and overall survival (OS) were reported using Kaplan-Meier estimation with 95% confidence bands, and differences in outcomes between FA and DA compared using log-rank tests...
April 18, 2024: Pediatric Blood & Cancer
#17
JOURNAL ARTICLE
Lucia Pedace, Simone Pizzi, Luana Abballe, Maria Vinci, Celeste Antonacci, Sara Patrizi, Claudia Nardini, Francesca Del Bufalo, Sabrina Rossi, Giulia Pericoli, Francesca Gianno, Zein Mersini Besharat, Luca Tiberi, Angela Mastronuzzi, Elisabetta Ferretti, Marco Tartaglia, Franco Locatelli, Andrea Ciolfi, Evelina Miele
In vitro models of pediatric brain tumors (pBT) are instrumental for better understanding the mechanisms contributing to oncogenesis and testing new therapies; thus, ideally, they should recapitulate the original tumor. We applied DNA methylation (DNAm) and copy number variation (CNV) profiling to characterize 241 pBT samples, including 155 tumors and 86 pBT-derived cell cultures, considering serum vs serum-free conditions, late vs early passages, and dimensionality (2D vs 3D cultures). We performed a t-SNE classification and identified differentially methylated regions in tumors compared to cell models...
April 18, 2024: NPJ Precision Oncology
#18
JOURNAL ARTICLE
Emily R Christison-Lagay, Erin G Brown, Jennifer Bruny, Melissa Funaro, Richard D Glick, Roshni Dasgupta, Christa N Grant, Abigail J Engwall-Gill, Timothy B Lautz, David Rothstein, Ashley Walther, Peter F Ehrlich, Jennifer H Aldrink, David Rodeberg, Reto M Baertschiger
BACKGROUND: Tunneled central venous catheters (CVCs) are the cornerstone of modern oncologic practice. Establishing best practices for catheter management in children with cancer is essential to optimize care, but few guidelines exist to guide placement and management. OBJECTIVES: To address four questions: 1) Does catheter composition influence the incidence of complications; 2) Is there a platelet count below which catheter placement poses an increased risk of complications; 3) Is there an absolute neutrophil count (ANC) below which catheter placement poses an increased risk of complications; and 4) Are there best practices for the management of a central line associated bloodstream infection (CLABSI)? METHODS: Data Sources: English language articles in Ovid Medline, PubMed, Embase, Web of Science, and Cochrane Databases...
March 27, 2024: Journal of Pediatric Surgery
#19
JOURNAL ARTICLE
Joaquin Garcia-Solorio, Juan Carlos Núñez-Enriquez, Marco Jiménez-Olivares, Janet Flores-Lujano, Fernanda Flores-Espino, Carolina Molina-Garay, Alejandra Cervera, Diana Casique-Aguirre, José Gabriel Peñaloza-Gonzalez, Ma Del Rocío Baños-Lara, Ángel García-Soto, César Alejandro Galván-Díaz, Alberto Olaya-Vargas, Hilario Flores Aguilar, Minerva Mata-Rocha, Miguel Ángel Garrido-Hernández, Juan Carlos Solís-Poblano, Nuria Citlalli Luna-Silva, Lena Sarahi Cano-Cuapio, Pierre Mitchel Aristil-Chery, Fernando Herrera-Quezada, Karol Carrillo-Sanchez, Anallely Muñoz-Rivas, Luis Leonardo Flores-Lagunes, Elvia Cristina Mendoza-Caamal, Beatriz Eugenia Villegas-Torres, Vincent González-Osnaya, Elva Jiménez-Hernández, José Refugio Torres-Nava, Jorge Alfonso Martín-Trejo, María de Lourdes Gutiérrez-Rivera, Rosa Martha Espinosa-Elizondo, Laura Elizabeth Merino-Pasaye, María Luisa Pérez-Saldívar, Silvia Jiménez-Morales, Everardo Curiel-Quesada, Haydeé Rosas-Vargas, Juan Manuel Mejía-Arangure, Carmen Alaez-Verson
BACKGROUND: Recurrent genetic alterations contributing to leukemogenesis have been identified in pediatric B-cell Acute Lymphoblastic Leukemia (B-ALL), and some are useful for refining classification, prognosis, and treatment selection. IKZF1plus is a complex biomarker associated with a poor prognosis. It is characterized by IKZF1 deletion coexisting with PAX5 , CDKN2A/2B , or PAR1 region deletions. The mutational spectrum and clinical impact of these alterations have scarcely been explored in Mexican pediatric patients with B-ALL...
2024: Frontiers in Oncology
#20
JOURNAL ARTICLE
Juan Carlos Núñez-Enríquez, Rubí Romo-Rodríguez, Pedro Gaspar-Mendoza, Gabriela Zamora-Herrera, Lizeth Torres-Pineda, Jiovanni Amador-Cardoso, Jebea A López-Blanco, Laura Alfaro-Hernández, Lucero López-García, Arely Rosas-Cruz, Dulce Rosario Alberto-Aguilar, César Omar Trejo-Pichardo, Dalia Ramírez-Ramírez, Astin Cruz-Maza, Janet Flores-Lujano, Nuria Luna-Silva, Angélica Martínez-Martell, Karina Martínez-Jose, Anabel Ramírez-Ramírez, Juan Carlos Solis-Poblano, Patricia Zagoya-Martínez, Vanessa Terán-Cerqueda, Andrea Huerta-Moreno, Álvaro Montiel-Jarquín, Miguel Garrido-Hernández, Raquel Hernández-Ramos, Daniela Olvera-Caraza, Cynthia Shanat Cruz-Medina, Enoch Alvarez-Rodríguez, Lénica Anahí Chávez-Aguilar, Wilfrido Herrera-Olivares, Brianda García-Hidalgo, Lena Sarahí Cano-Cuapio, Claudia Guevara-Espejel, Gerardo Juárez-Avendaño, Juan Carlos Balandrán, Ma Del Rocío Baños-Lara, Mariana Cárdenas-González, Elena R Álvarez-Buylla, Sonia Mayra Pérez-Tapia, Diana Casique-Aguirre, Rosana Pelayo
The main objective of the National Project for Research and Incidence of Childhood Leukemias is to reduce early mortality rates for these neoplasms in the vulnerable regions of Mexico. This project was conducted in the states of Oaxaca, Puebla, and Tlaxcala. A key strategy of the project is the implementation of an effective roadmap to ensure that leukemia patients are the target of maximum benefit of interdisciplinary collaboration between researchers, clinicians, surveyors, and laboratories. This strategy guarantees the comprehensive management of diagnosis and follow-up samples of pediatric patients with leukemia, centralizing, managing, and analyzing the information collected...
2024: Frontiers in Oncology
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